Family‐centred care interventions for children with chronic conditions: A scoping review

Abstract Introduction Children with chronic conditions have greater health care needs than the general paediatric population but may not receive care that centres their needs and preferences as identified by their families. Clinicians and researchers are interested in developing interventions to improve family‐centred care need information about the characteristics of existing interventions, their development and the domains of family‐centred care that they address. We conducted a scoping review that aimed to identify and characterize recent family‐centred interventions designed to improve experiences with care for children with chronic conditions. Methods We searched Medline, Embase, PsycInfo and Cochrane databases, and grey literature sources for relevant articles or documents published between 1 January 2019 and 11 August 2020 (databases) or 7–20 October 2020 (grey literature). Primary studies with ≥10 participants, clinical practice guidelines and theoretical articles describing family‐centred interventions that aimed to improve experiences with care for children with chronic conditions were eligible. Following citation and full‐text screening by two reviewers working independently, we charted data covering study characteristics and interventions from eligible reports and synthesized interventions by domains of family‐centred care. Results Our search identified 2882 citations, from which 63 articles describing 61 unique interventions met the eligibility criteria and were included in this review. The most common study designs were quasiexperimental studies (n = 18), randomized controlled trials (n = 11) and qualitative and mixed‐methods studies (n = 9 each). The most frequently addressed domains of family‐centred care were communication and information provision (n = 45), family involvement in care (n = 37) and access to care (n = 30). Conclusion This review, which identified 61 unique interventions aimed at improving family‐centred care for children with chronic conditions across a range of settings, is a concrete resource for researchers, health care providers and administrators interested in improving care for this high‐needs population. Patient or Public Contribution This study was co‐developed with three patient partner co‐investigators, all of whom are individuals with lived experiences of rare chronic diseases as parents and/or patients and have prior experience in patient engagement in research (I. J., N. P., M. S.). These patient partner co‐investigators contributed to this study at all stages, from conceptualization to dissemination.

with chronic conditions. 7,80][11] In paediatrics, family-centred care recognizes that the family is the primary unit of support for a child's health care management and overall development. 124][15][16][17] Meaningful engagement of patients and families in the co-design or study of interventions that affect them is consistent with a family-centred care approach and may improve intervention effectiveness and family satisfaction. 18,19nfronted with a broadly defined topic, those interested in developing interventions to make care more family-centred, such as policymakers, clinicians, and researchers and representatives of health care systems, may seek to learn more about, and build upon, framework. 20The authors described four main types of familyoriented interventions for parents: education about disabilities and training to administer therapy; support groups; psychosocial interventions (e.g., counselling, coaching); and provision of information about disability and available resources. 20Their proposed framework for family-oriented services included six domains: information resources; education services; training/instruction services;, support groups; psychosocial services; and service coordination.
Building on King and colleagues' review, our focus for this scoping review was somewhat broader.Our overall aim was to identify and characterize recently published family-centred health care interventions across a range of health care settings (including but not limited to rehabilitation settings) that sought to improve experiences with health care for children with chronic conditions and their families.More specifically, we sought to describe: (i) how the With respect to domains of family-centred care, we used an existing framework, the Picker principles, 21,22 as a guide.This set of principles was developed by the US Picker Institute to advocate for the needs and preferences of patients and families in health care interactions. 23[26] 2 | METHODS Scoping reviews are ideal for characterizing a broad landscape of literature on a particular topic, aligned with our goal. 27,28Our scoping review methods followed the Joanna Briggs Institute (JBI) Methodology for Scoping Reviews 29 and our published protocol (available at https://osf.io/cjyd9/?view_only=0077822cf9ef424290651ed7b5c8 0177).We report our review in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRIS-MA) extension for scoping reviews (PRISMA-ScR) (Appendix S1). 30

| Patient and family engagement
Family caregivers of children with chronic conditions interact frequently with the health care system, becoming experts in their children's care needs. 31,32Their partnership and engagement in research, especially about family-centred care, can be expected to facilitate research questions that are more meaningful and relevant to children and their families and contribute to successful care improvement efforts. 33,34This study was co-developed with three patient partner co-investigators, all of whom are individuals with lived experiences of rare chronic diseases as parents or patients and have prior experience in patient engagement in research (I.J., N. P., M. S.).These patient partner co-investigators contributed to this study at all stages, from conceptualization to dissemination.Specifically, they participated in decision-making about the concept of the review and its scope, contributed to the interpretation of the extracted data and critically reviewed the final manuscript.

| Search strategy and information sources
An experienced medical information specialist (B.S.) developed and tested the search strategies through an iterative process in consultation with the research team, including review of a preexisting, extensive strategy covering a range of similar chronic conditions. 35Another senior information specialist peer reviewed the strategies before execution using the PRESS Checklist. 36Suggestions were reviewed and if applicable, incorporated into the final strategy.

| Eligibility criteria
To be eligible for inclusion, articles had to describe a family-centred care intervention for children 12 years and younger with chronic conditions and/or their families (Table 1).Operationalization of these concepts is described below.These criteria were designed to balance a desire to include a wide range of recently published family-centred care interventions.

| Study design types
Primary studies of any design were eligible except for case series and primary study designs with fewer than 10 participants, including case studies.Theoretical articles describing family-centred care models or frameworks that have not yet been implemented were included if the article had been peer-reviewed.Clinical practice guidelines were also included.Systematic and other secondary review studies were excluded as we anticipated that they would be repetitive.

| Population
We included studies where the target of the intervention was children aged 12 years or younger with chronic conditions and/or their families.Interventions or studies that targeted solely adolescents (aged 13-18 years) were excluded as their health care needs are often distinct from those of younger children.'Chronic conditions' were defined as requiring an elevated number or intensity of interactions with the health care system relative to the general paediatric population, expected to be required for 1 year or more.

| Concept
We included articles that described family-centred care interventions (activities, strategies or policies). 38Family-centred care was defined as an approach to the planning, delivery and evaluation of health care that was grounded in mutually beneficial partnerships among health care providers, patients and families. 13We considered an intervention to be family-centred if: the main objective of the intervention was to improve health care experiences (including timely access to care or treatment, coordination of care, communication with providers and emotional support) for the population; and both (i) health care providers and/or administrators and (ii) primary caregivers of children were actively engaged in the development or research of the intervention (e.g., in the study of implementation of the intervention).With respect to this latter criterion, we considered engagement of primary caregivers and providers/administrators to be critical given their roles as knowledge users positioned to benefit from or to implement respectively, interventions to improve familycentred care.Included articles had to describe the intervention's activities or processes or cite other articles accessible to the authors that did.We excluded: screening or preventive interventions targeting children that did not have a pre-existing, chronic condition; clinical drug treatment regimens, which are typically not generalizable and do not directly target improvement of health care experiences; interventions targeting transition from paediatric to adult health care services; institutional networks and partnerships; and existing services (longstanding, geographically widespread programmes that were not new interventions to improve care).

| Context
We included interventions that addressed physical or mental health care for a child, were implemented in any setting (e.g., health care, school, home) for acute or nonacute care and involved any health care professional (e.g., physician, nurse, rehabilitation therapist).

| Other
We excluded protocols, conference abstracts, theses and dissertations, commentaries and letters.We included articles irrespective of the outcomes studied.Only articles published in or after 2019 were included.As many grey literature websites and documents did not report a publication date, we chose to include all undated grey literature.Only articles published in English were included.

| Study selection
In stage 1 of a two-stage screening process, we used a liberal accelerated approach. 39Two reviewers (among A. J. C., Z. A., R. I.) independently screened the titles and abstracts of identified records.
A record was retained if at least one reviewer positively assessed that it met the eligibility criteria or was uncertain about its inclusion.For the grey literature, one author (A.J. C. or Ammar Saad) performed stage 1 screening.
In stage 2, the full-text articles of all records retained in stage 1 were obtained.If an article could not be obtained electronically free of charge, we contacted the corresponding or lead study author by email up to two times and excluded the article if a copy could not be obtained.Two reviewers (among A. J. C., Z. A., R. I., Ammar Saad) independently screened all full-text articles and grey literature in duplicate.We resolved disagreements by discussion and consensus; an arbitrator (B.K. P.) resolved disagreements that could not be resolved by the reviewers.Clinical experts (P.C., M. K.) provided checklist. 40We charted information about the reporting of each intervention including: the care delivery setting involved, the authors' stated objectives and rationale for, or theory grounding, each intervention, and the intervention's components (specific activities/processes).Our adapted checklist did not include TIDieR checklist items #11 and #12, about planned and actual fidelity to the intervention, as we did not aim to assess intervention effectiveness.

| Synthesis
Using the data charted, we synthesized the development of the interventions by activity type (e.g., feedback seeking, pilot testing) and groups engaged in development (e.g., caregivers, children, health care providers).We also summarized the broad care delivery settings (i.e., inpatient, outpatient, community) and the chronic conditions of the target populations, grouping by pathophysiological manifestations and symptoms.
We summarized the activities/processes and objectives of each intervention and identified which among eight family-centred care domains the intervention sought to address 21,22 (definitions, Appendix S4).For the latter characterization, we used an iterative coding approach, where one reviewer (Ammar Saad) coded and a second team member (A.J. C.) verified.We summarized the common types of intervention approaches within each family-centred care domain.

| Study selection
After deduplication and removal of records published before 2019, we screened 2882 records by their titles and abstracts; of those, 369 full-text reports were assessed for eligibility (Figure 1).We further

| Study characteristics
Table 2 and Appendix S5 describe the 63 included articles, the majority of which reported on primary research studies (n = 57; 90.5%).The most common primary study design was quasiexperimental (n = 18; 28.6%), followed by randomized controlled trial (RCT) (n = 11; 17.5%).The sample size of the 57 included primary research studies ranged from 10 (per our inclusion criteria) to 6,259 participants, with almost a third reporting a sample size of 10-30 participants (n = 18/57; 31.6%).Seventy-nine percent of articles described studies conducted in (or, if not stated, country of affiliation of lead author) North America (n = 40; 63.5%), as well as Europe (n = 6; 9.5%) and the United Kingdom (n = 4; 6.3%).Interventions were delivered in-person (n = 23; 37.7%), virtually (n = 13; 21.3%) or using both modes (n = 18; 29.5%).Seven interventions (11.5%) did not specify a delivery mode.29.5%) (Figure 3).There may also be a paucity of interventions addressing the needs of siblings of children with chronic conditions.Although 10 interventions described 'families' as a target of their activities, we identified only one intervention in which activities directly targeted siblings. 47o additional interventions included advising or educating caregivers to be aware of siblings' emotional needs after a diagnosis or medical event (data not shown). 56,88Healthy siblings of children with chronic conditions may be more likely to experience adverse psychological effects, especially if the condition is more severe. 133re interventions that directly address the needs of siblings are an important part of current efforts to make care more family-centred.

| Coordination of care among providers and with the family
Care coordination is a particularly important aspect of family-centred care for children with chronic conditions, who often have ongoing relationships with care providers, regularly interact with multiple facets of the health care system beyond primary care, and need daily at-home care management. 134Health care systems often perform poorly in meeting the needs of this population, with fragmented or siloed institutions and providers and a lack of mechanisms for coordination of care and appointments and sharing of information across providers and systems. 6,134,135Preferred approaches to health care coordination for children with chronic conditions that have been described include team-based organization of care, designation of a care coordinator, digital means of information sharing among providers, care plans and patient registries to track and monitor patients. 136,137   We implemented two deviations from our published protocol: (i) we described the planned review as 'rapid' in our protocol but at this reporting stage we believe that the 2-year timeframe needed to complete it renders that description inaccurate; and (ii) we changed the eligibility criteria regarding publication date for feasibility reasons.
With respect to (ii) family-centred care is a broad concept,
Our strategy was structured and designed to emphasize sensitivity over specificity (Appendix S2).Using the multifile option and deduplication tool on the OVID platform, we searched Ovid MED-LINE ® ALL, including Epub Ahead of Print, In-Process & Other Non-Indexed Citations, Embase Classic+Embase, APA PsycInfo, and the following EBM Reviews Databases: Cochrane Central Register of identified 127 records through grey literature searches, and after deduplication and removal of records published before 2019, we retrieved and assessed 59 full-text reports for eligibility (Figure1).A total of 63 reports were included in this review, representing 61 unique interventions. Furthemore, we consulted additional publications, which were cited in the eligible reports, for additional details about 23 of the 61 included interventions during our data charting.

For the majority of the 61
Figure 2A and Appendix S5).Among interventions where authors described who was involved in development, caregivers (e.g., parents) were engaged in developing about half the interventions (n = 31; Figure 2B and Appendix S5).A similar proportion described involving health care providers (n = 28), whereas child patient involvement was described in the development of 15 interventions.

F 1 |
I G U R E 1 Preferred Reporting Items for Systematic reviews and Meta-Analyses flow diagram of study screening and selection.by a multidisciplinary team in the patient's medical home or via a single point-of-care (15/28 interventions).We identified more types of intervention approaches to the access to care domain (n = 6) than any other domain.4 | DISCUSSION 4.Summary of findings This scoping review aimed to identify and characterize recently described interventions designed to make care more family-centred for children with chronic conditions and their families, so that practice and research can build on a synthesis and an understanding of how interventions have been developed to date, the domains of family-centred care that these interventions have addressed and the key components of the interventions that have sought to address those domains.This was an important evidence gap, particularly since family-centred care is widely agreed to be important but is broadly defined in the literature, rendering it challenging to understand which approaches have been developed to address specific domains, paediatric chronic disease patient populations and settings.We identified 63 articles published over the course of fewer than 20 months that described family-centred care interventions for children with chronic conditions.They encompassed a broad range of specific aims related to improving aspects of family-centred care, activities and methods.Confirming family-centred care as a multifaceted concept, nearly half of the included interventions addressed at least four family-centred care domains while only a few addressed a single domain.Communication and information provision, access to care and family involvement were the most common domains addressed by included interventions' aims and activities, though all eight of the domains that we considered, based on the Picker principles of patient-centred care, were addressed by multiple interventions.Clear and comprehensive reporting of interventions is important40 ; therefore, we assessed the reporting of included interventions using the TIDieR checklist (Appendices S6 and S7).In our assessment, reporting of many details recommended by the TIDieR checklist was strong, with >85% of studies adequately reporting an intervention name; a rationale, theory or goal of the intervention's elements; activities or procedures; facilitators or providers; and setting.We note that reporting of the intervention's goals and activities were among the inclusion criteria for our review, which likely led to a sample of interventions that were relatively better defined.We identified room for improvement in the reporting of intervention materials, mode of delivery, intervention timing and duration, planned tailoring and actual modifications.Family and patient co-design of interventions, particularly those aiming to be family-centred, broadens the range of ideas and perspectives in design and may improve intervention effectiveness.
of interventions.We anticipated a higher proportion as engagement of health care providers or administrators and of families in the development or research of an intervention was an inclusion criterion.Among the interventions that described such involvement in development, caregivers and child patients were reported to have been engaged in the development of 72.7% and 34.8% of the interventions, respectively.There is still room for improvement in engaging these groups in the development of family-centred care interventions and the reporting of such engagement, particularly children, even with consideration of age and developmental capacity.

43 ]
Our review indicates that recent interventions adhere to those recommendations, as we identified four common approaches to address care coordination: medical home or teambased models; designation of individuals or teams to coordinate aspects of care; processes for sharing information among providers; and using technology to enhance coordination.The most common of these was the medical home or team-based model, which was examined or described by nearly a quarter of the interventions we reviewed.Such models aim to provide comprehensive, coordinated and accessible care, often through primary care, 138 though interventions we reviewed that addressed care coordination did so in both hospital and community settings.Several interventions identified one or more care team members to conduct care coordination activities or streamline a referral process.When these team members liaise directly with families, such points of contact also provide an opportunity for relational continuity, building familiarity and trust between families and health care teams.This continuity is F I G U R E 2 (A) Activities involved in intervention development.Feedback-seeking activities: Quantitative and qualitative data collection of opinions and feedback from contributor groups on content or design.Piloting/refining: Feasibility testing and pilot implementation of interventions.(B) People and entities engaged in intervention development alongside researchers.Caregivers: Parents, caretakers and legal authority representatives.Subject/content: Clinical or intervention design expertise.Others: Allied health professionals, information technology specialists, insurers, teachers, nongovernmental organizations, community advocates and governmental health entities.T A B L E 3 Characteristics of included interventions.Interventions delivered in or from outpatient clinical settings (n = 22) [41] Website to prepare parents to take part in decision-making during their consultation with a rehabilitation physician; helps families identify their needs + Collaborative meetings between families + therapists, at clinic or home (family preference), to codevelop + implement child development intervention, including family needs, activities + medical home care coordination programme that includes caregiver education (guidebook), team-based care + clinical care plan Children, caregivers T: U/S D: U/S M:

4. 4 |
Strengths and limitationsThe high number of published articles returned in our search highlighted a need for a comprehensive, good-quality review about family-centred care interventions to provide policymakers, clinicians and researchers engaged in health care improvement for children with chronic conditions and their families with a useful summary of the literature.To create this resource of family-centred care interventions, we aimed to identify and describe the scope of existing family-centred care interventions, mapping their component activities and processes within established domains.We used rigorous methods, particularly in the search strategy and screening and extraction of articles, for example, including multiple reviewers and extractors to minimize bias; and we relied on widely accepted principles of family-centred care to synthesize our findings.

| 15 of 24 F
Abbreviations: CPG, clinical practice guideline; FU, follow-up; HCP, health care provider; IP, in-person; NICU, newborn intensive care unit; U/S, unspecified.a 'Depends' defined as: Intervention timing and/or duration depends on the needs and preferences of the recipients and/or their families their pain symptoms and location to providers Incorporate pain management strategies (e.g., medications, relaxation training) into the care plan narrow the eligibility to publications since 2019 enabled us to identify interventions that address the most current contexts and needs.The exclusion of articles published before 2019, as well as those published in languages other than English, may introduce selection bias into our review. 149Interested readers may refer to Appendix S9 for a list of excluded non-English articles.The English-language eligibility criterion also likely affected the geographic distribution of articles included in this review: more than two-thirds of articles described interventions implemented in North America or the United Kingdom.Finally, this review focused on a broad topic, interventions to improve family-centred care.We strove to identify and include interventions of interest but some relevant interventions may have been missed or described in article types (e.g., commentaries) that were ineligible for our review.5| CONCLUSION As recognition of the unmet needs of children with chronic conditions grows, so does interest in improving family-centred care.The identification of 61 family-centred care interventions for children with chronic conditions, described in articles published 2019 to mid-2020, demonstrates that this is an active area of research.The breadth of the concept of family-centred care may present a challenge for individuals and groups interested in developing and evaluating interventions who want to build on previous work.This review is a concrete resource for health care providers, administrators and researchers, providing an inventory of interventions categorized by family-centred care domain, setting and population, that describes the types of activities and processes that have been developed and/or implemented recently.It serves as a foundation for those engaged in practice and/or research to improve health care for children with high needs and their families by highlighting interventions that centre the needs of children and their caregivers, and to potentially advocate to governments and funding agencies for the resources to improve that care.The many interventions that have been the subject of RCTs and quasiexperimental designs underscores an opportunity for future systematic reviews to evaluate the effectiveness of interventions for a subset of domains, populations or settings, using our inventory as a starting point.Finally, we have highlighted two areas where future research on the development of future family-centred care interventions may be improved: (1) involving patients and families in the development process and (2) improving the transparent reporting of intervention development and implementation, particularly with respect to clarifying aims and processes of engaging with patients, caregivers and providers.

Table 4
describes types of approaches taken to addressing each family-centred care domain, with intervention examples.For example, care coordination was most commonly addressed by adopting a team-based approach to care delivery, in which services are provided Characteristics of included articles.
18,19For close to 30% of included interventions, authors did not report or cite information on the groups engaged in the developmentT A B L E 2a Primary studies only.